Case Studies

PLUNGING RANULA: A CASE REPORT AND A LITERATURE REVIEW

NEII. E.I. IANGLOIS. MB BCHIK, MA, AND PKALHAI) KOI.HE, FRCS

The plunging runula i.5 (1 mucows ratrclz~rc.tntion cyst of the .suhlingual where ;I ttissectiou of the cyst re~xdect it 10 lx attached to the
gland. It i.s .tlightly more common inJemc~le.s, shoua no .side pwfkwnce. suhrn~undit,t~l~ir &d. The cyst watt with the adherent salivar\~
and is more prel&ent iu the .second ccnti third dec~1e.s oj’ l{fe. It gland W:I~ excised and terra&line powder installed into the
t~pirull~ mt~ n{fr.st.c MS o pnin1e.s.s. nonmobile .swlli ng in the rwk cavity to ohtileratc it. At rev’iew 4 months later the swelling
and in fburofjizw
ems i.s ccssociated with au intrwrtll ran ula I)) had I-rcurrecl agairi.
swelling. If therr is no hi,story oJ an orctl rclnula the clinicctl diqno.sis
is di’cultl cold it mq be leji to the wporting pathologi.st to gjvr the PATHO1,0(;1
rorrrrt diqnosis. The hi.stologic ~ppeur(~nce i.s chnr~L~t~,~-i.stirull~ of
‘t‘hc specinien receivect consisted of an irregular portion
o cyst. dezloid of rpithelium or endothelium, with CLvclsruln~- fihro-
of tissue, one surface of which !+as smoorh, suggesting it was
co~~nectiz~e ti.csl;e wll contnining .somr chronic inflammctto~~ cel1.c
ai1 otmiecl cyst. Histologic csaniination confirmecl the presence
and mar rvphage.s .stuffed with m rtcin. Thr correct dmgnosi.s i.s ~.S.SFII-
of a cystic structure. The w~dl was colnposect of loose. cascular-,
tiol Jijr the most t$rtiur treatment. which i.s rscision of the .subliugunl
Copyright 0 1992 by W.A.
fibroconnec~ive tissue rictl in connective tissue mucin (Fig 2).
gland. Hr:v P.~~‘HoI. 23: 1 X5-IXM.
Snu nders Corn/xc n?l
On the inner surface were niacrophages containing mucin that
stained positivetv with mucicarmine and diast;~s~-digeste~t ptl-
Ranutas are mucous retention cysts of the sublingual riodic acid-Schift. (Fig 3). Else of i~r~munohisto(.tle~~~isrI-y (XI-
gland. Whereas the oral ranuta is relatively common and pre- tibodies to CAM 5.2. AE I /AE3. and factor VIII) ctemonsrrated
sents as a cyst in the mouth, the plunging ranula is rare and the absence c’f an endothetiat or epithetiat lining. Within the
manifests itself as a mass in the neck with or without an as- outer cvst watt portions of a mixed (mutinous anct serous)
sociated oral lesion. If there is no intraoral component, clinical salivary gta~id wel-e presenr.
suspicion may be low and the pathologist may he misled by
the histologic appearance.’ Recognition of the diagnosis of
DISCL’SSION
plunging ranuta is essential for the correct treatment of these
lesions. We report a case of a 9-year-old girt and review the In cxbntrast I0 ttiv oral randa, the t~tunging rxiiit;i is a11
English literature to discuss the epidemiology and pathology unco1nmon lesion. To manifest as a IKISS in the neck without
of these lesions. ;m oral lesion. AS in this case, is even more I-are.‘The difttrential
clinical diagnosis includes I!Illphadenopathy. abscess, thyrog-
CASE REPORT tossat dust cyst, dermoict or epidel-maid cyst, taryngocete, ti-
A Svear-old girt complaining of a large cystic swelling in poma. hemangioma. cervical thymic cyst, cysts of the paralhy-
the left side of her neck was referred to a dental surgeon h) roid or thyroid gland. or Iumor.‘.”
her general practitioner in May 1990. No dental pathotoby 4 review of the case reports of plunging ranutas in the
was identified and the patienr was referred to the ear, nose, English tirerature revealed a mate to female ses ratio of I : I .S
and throat department. The swelling had been present fo1 and no significant side preference. Presentation was most
approximately 3 months; it exhibited no change with eating, co~n~non in the second and third decades of life. with an age
chewing, or swallowing. There had been no discharge. On range of 3’ IO 61 ’ years. Typically, the history was of a painless
examination a nontender, diffuse, cystic mass could he seen mass that did not vaq in size with eating, chewing, or swat-
and felt in the left submandibular area. The tentative diagnosis lowing. However, pain was an occasional feature.‘.” Unusual
of hranchiat cyst or cystic hygroma was made. In October 1990. presentations included rapid enlargement following infection”
the patient underwent a left neck exploration, A muttitobutated and varying size with the valsatva maneuver.’ Asynchronous
cystic lesion, deep to the ptatysma and wrapped around the involvement of both sides also has been reported.8 The majority
submandibular gland. was discovered. The cyst extended into of patients presented within 3 months of occurrence of the
the floor of the mouth. lateral pharyngeal watt, and tonsils, swelling, but the reported range was 24 hours” to I8 years.”
reaching the skull base, where the internal carotid artery and The majority of masses were 4 to 10 cm in size and lay within
stytoid process were palpable. The cyst ruptured during dis- the submandibular area of the neck, but extension to the sub-
section, but as much as possible was excised. The diagnosis of mental region and both sides of the neck,‘“,” to the base of
plunging ranuta was made on pathologic grounds. the skutt,“~” to the retropharynx,” to the clavicle,‘,” and even
By December 1990, the swelling had recurred (Fig 1). from both ears to the sternum” has been reported. The re-
The patient was referred to the department of plastic surgery ported cases included comments concerning the intraoral ap-
pearance of the masses: 45% presented first with an oral swett-
From the Department of Pathology, University of Aberdeen, ing or ranula, 34% had an associated oral mass at presentation,
Aberdeen, Scotland; and the Department of Surgery, Aberdeen Royal and 21% had only a swelling in the neck.
Infirmary, Aberdeen, Scotland. Accepted for publication June 16. The histologic appearance is typically that of a cyst, devoid
1992. of epithetiat lining, with the wall composed of vascular fihro-
Rq words: ranula. plunging. cervical.
connective tissue resembling granulation tissue with mucin and
Address cot-respondence and reprint requests to Neil E.I. Lan-
glois, MB BChir. MA, Department of Pathology, University Medical foamy macrophages. Partial epithetial lining, however, has been
Buildings, Foresterhill, Aberdeen AB9 2ZD. Scotland. recorded.14 Batsakis and McCIatchey” note that the histologic
Copyright 0 1992 by W.B. Saunders Compaq appearance changes with time from a loose. vascutarized con-
0046~8177/92/2X1 l-0018$5.00/0 nective tissue with an abundance of mucin to a predominantly

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 CASE STUDIES

FIGUR !E 1. Profile of the patient showing the scar

from tTer firs?- operation and the recurrence of the
plunging ranula

FIGURE 2. A typical area

from the plunging ranula
showing the cystic space
(center and left) bound by
vascular connective tissue
devoid of epithelial lining.
 HUMAN PATHOLOGY Volume 23, No. 11 (November 1992)

FIGURE 3. Higher-power
view to demonstrate the
foamy macrophages that
line the cyst and stain posi-
tively with diastase digested
periodic acidschiff

dense, well-\,asclllal-izecl tissue with .t paucity ofntuciti. Plung-

ing ranulas also have been associaLed with vascular
tiialforniatior~s’” and dermoid cvsts.“’
From analysis of the sect&m within the ranula it appears
that their origin is due to a extravasation of saliva from the
sublingual gland forming a pseudocyst without epithelial lin-
ing.” It has been suggested that leakage may either be due to
trauma,’ obstruction of the salivary duct,17 or salivary duct
anomaly.” The plunging ranula may arise in the neck by three
possible mechanisms: the sublingual gland tnay project through
the mylohyoid” to provide an origin.‘” the cyst may penetrate
through the mylohyoid to join the sublingual gland in the
mouth,‘.‘” or a duct from the sublingual gland may join the
submaxillary duct”‘~‘” and give rise to ranula in continuity with
the submandibular gland or its duct.1’,2’ The latter explanation t 1. Hitchin AD: Raluta. Br Dent J XfJ::325-327, 1!btfi
is favored in this case as the ranula was adherent to the sub- IL’. van den Akker HP, Bays RA. Becker- AK: Plunging or wrbicat I-anula:
maxillary gland, but recurred after the gland was excised. A review of the litcratwe and a’report of 4 caws. J Maxillofa Surg 6:286-29X
Reviews of surgical management of this lesion underline 197X
the necessity of removal of the sublingual gland rather than
the excision of the cyst itself. Typically, more than one oper-
ation is required to effect a cure, the highest success rate being
achieved by excision of the sublingual gland.‘,‘2,2’.L’2
This case revealed several interesting features: the pre-
sentation was solely of a swelling in the neck, it was found to
extend to the base of the skull, and it appeared to take its
17. Harrison JD, (;a~-rer JR: Hisrologicat effrcts 01 ductal liptiun of the
origin from the submandibular gland. However, the pathologic salivary glands of the ~1. J Pathot 11X:215-254. 1975
appearance was typical, which enabled the correct diagnosis. 18. C:anghran GRL: Mylohyoid boutoniere and sublingual bouton. J Anat
Thus, the plunging ranula is a pseudocyst of the sublingual !l7:5ti5-568, I!163
gland and should be treated as such. I!). RI-idger AG, Carter P. Br-idgrr C;P: Plunging rmuta: Literature revieu
mtl rl qort of three case% Aust N % J Surg 59345.948. 1989
20. Williams PL. Warwick R, Dyson M. et at (eds): Splanchnoto~~, in Gray’s
Anatomy. (37th ed). Edinburgh. UK. Churchill Livingstone. 19X9. p I?93
21. Par&h D. Stewart M. Joseph C. er at: Plunging ranuta: A repel-c of
REFERENCES three casts and a review of the literature Rr J Surg 74:307-309. 19X7
1. McClatrhey KI). Haley-AppelhlattN. %arho RJ. ct al: Piu,,gir,g I-arrula. 22. Yoshimura Y, Sughara T, Matsuura R: Plunging nnuta: A report of
Oral Surg Oral Med Oral Pathol 5i:1OX-4 I”, 198-l two cases and P review of the tierature. Quintessence Int ?0:435-437. 1989

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